August 2013 Vol. 15 No. 2 [Link].

org/ppl
Editor: Carl L. von Baeyer, PhD, [Link]@[Link] Associate Editor: Deirdre E. Logan, PhD
© 2013, Special Interest Group on Pain in Childhood, International Association for the Study of Pain®

Commentary
Pain in children with Autism Spectrum Disorder:
Experience, expression, and assessment
Amanda K. I. Knoll, C. Meghan McMurtry and Christine T. Chambers
Introduction have occurred in the absence of definitive research
findings. Arguing against pain insensitivity, Nader
Historically, children with developmental et al. (2004) found that during venipuncture,
disabilities have been excluded from pain research children with ASD displayed overall facial pain
(Breau et al., 2006) despite comorbidities that responses comparable to typically developing
increase risk of pain (Bottos & Chambers, 2006). children and greater facial reactivity during the
Children with developmental disabilities and needle phase. The objectives of this commentary are
communication impairments may be particularly at to examine: (a) pain expression and experience in
risk as pain in individuals with communication ASD, (b) challenges in pain assessment, (c) and the
impairments is frequently ignored and ineffectively role of socio-communicative deficits and pain
managed (Hadjistavropoulos et al., 2001; context.
Oberlander & Symons, 2006; Craig, 2009). One
such population is children with Autism Spectrum Pain expression and experience
Disorder (ASD). In the 5th edition of the Diagnostic The way that a person expresses pain can give
and Statistical Manual of Mental Disorders (DSM- insight into his/her pain experience, but these are
5), ASD is conceptualized as a disorder involving not identical constructs (Craig, 1986). Pain
difficulties in social communication and interaction expression involves a person’s observable response
as well as “restricted, repetitive patterns of behavior, to a noxious stimulus, including pain behaviors,
interests, or activities”; this latter criterion includes whereas a person’s pain experience is internal and
“hyper- or hyporeactivity to sensory input or includes severity of discomfort (Craig, 1986).
unusual interest in sensory aspects of the Children with ASD are often reported to respond
environment (e.g., apparent indifference to pain...)” differently to sensory stimuli compared to typically
(American Psychiatric Association, 2013). developing children; these responses are assumed to
Research on pain in children with ASD has reflect differences in sensory experience and/or
generally depended on parent report of child pain difficulties with sensory integration (Talay-Ongan
(Bottos & Chambers, 2006); caregivers have & Wood, 2000; Watling et al., 2001; Baranek, 2002;
frequently reported that children with cognitive Tomcheck & Dunn, 2007; Ashburner et al., 2008;
impairment and/or ASD experience pain differently Ben-Sasson et al., 2009; Klintwall et al., 2011).
than typically developing children (Fanurik et al., Unusual responses to sensory stimulation, including
1999; Militerni et al., 2000; Inglese, 2008). In a potential indifference to pain, are described in the
particular, there have been speculations that associated features and diagnostic
children with ASD display reduced sensitivity to conceptualizations of the disorder (McPartland et al.,
pain (e.g. Cascio et al., 2008; Messmer et al., 2008; 2012; American Psychiatric Association, 2013).
Tordjman et al., 2009)1. However, these contentions

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Some researchers also suggest that children with pain reactivity by their behavior compared to a
ASD may express pain differently, but the direction control group, individuals with ASD showed
and empirical bases of these differences are unclear significantly greater heart rate before, during and
(Militerni et al., 2000; Bottos & Chambers, 2006): after a venipuncture than the control group. An
studies report hypersensitivity (Nader et al., 2004; increase in heart rate is not necessarily specific to
Tordjman et al., 2009), hyposensitivity (Militerni et pain; anxiety or other forms of arousal may also
al., 2000), or both hypo- and hypersensitivity cause a change in heart rate. Hence, while
(Inglese, 2008). Children with ASD may differ from physiological measures such as heart rate do
typically developing children in general sensory provide a helpful index of possible pain reactivity,
sensitivity while having similar pain experiences they are also likely to be confounded by other types
(Minshew & Hobson, 2008). How do we of physiological arousal and/or distress (e.g.
understand these inconsistencies? Pain assessment anxiety).
method, socio-communicative deficits, and context Any proxy report of pain is likely to be based,
may all play a role. at least partly, on observable behaviors (Coll et al.,
2011). Much of the research on pain in children
Pain assessment
with ASD has been conducted using parent report
Pain assessment is commonly achieved due to communication challenges and difficulty
through self-report, observational, and/or proxy self-reporting pain (Bottos & Chambers, 2006).
methods. While self-report is frequently used with However, parents of children with ASD may not be
typically developing children, impaired reliable in providing proxy reports of their
communication skills may make self-report children’s pain, as their reports may be influenced
questionable for populations with ASD (Breau & by a variety of factors including information
Burkitt, 2009; Craig, 2009). Recent studies provided by health care professionals about the
(Minshew & Hobson, 2008; Bandstra et al., 2012) child’s sensation and reaction to pain (Breau et al.,
have gathered self-report from children and 2003). Parents of children with cognitive
adolescents with ASD, supplemented with other impairment have been shown to believe that their
measures (e.g. parent report, physiological children experience less pain than typically
measures). Findings indicate that some high- developing children (Breau et al., 2003); parents
functioning individuals with ASD may be able to may hold similar beliefs regarding children with
rate their own pain through self-report. However, ASD. These beliefs may influence pain ratings:
ASD is a spectrum: just because high-functioning Dickie and colleagues (2009) found that parents of
individuals with ASD are able to self-report their children with ASD were more likely than parents of
pain does not indicate that self-report is appropriate typically developing children to attribute sensory
for the entire population. responses as characteristic of ASD. Thus, parents of
Unfortunately, observational pain assessment children with ASD may report their child’s pain
can also be difficult due to idiosyncratic behaviors differently than parents of typically developing
associated with ASD and/or cognitive impairment children but whether this reflects true differences in
(e.g. atypical vocalizations, facial expressions) that pain experience is unclear. In fact, parent reports
may result in misestimates of pain by those have been shown to have no significant relationship
unfamiliar with the individual’s typical behavioral with self-reported and psychophysically-measured
responses (Fanurik et al., 1999; Bottos & Chambers, sensory sensitivity in children with ASD (Güçlü et
2006). Thus, different observers/caregivers may al., 2007). Similarly, during venipuncture, no
interpret pain behaviors differently (Coll et al., 2011; relationship was found between parent ratings of
Fanurik et al., 1999). Additionally, there may be a child pain and facial responses of children with
disconnect between observed pain behavior and ASD, whereas there was a moderate relationship for
physiological response: Tordjman et al. (2009) typically developing children (Nader et al., 2004).
found that while individuals with ASD were more Furthermore, strong negative correlations were
likely to be rated as demonstrating an absence of found between parental report of their child’s

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general pain sensitivity/reactivity and observed whether research findings on pain experience and
behavioral responses of children with ASD; no such expression reflect cognitive and developmental
relationship existed for typically developing delays in the research samples rather than
children (Nader et al., 2004). In sum, assessment characteristics associated with ASD per se.
method is important to consider when To understand inconsistencies in pain
understanding the experience and expression of pain expressions, pain context may also be critical
in this population. (Nader et al., 2004), particularly in populations with
ASD given inflexibility to changes in routines and
Role of socio-communicative deficits and
frequently comorbid anxiety (van Steensel et al.,
pain context
2011; American Psychiatric Association, 2013).
While it may be proposed that biophysical Pain behaviors in children with ASD may be more
differences result in (suspected) differences in pronounced in some contexts than others (Tordjman
sensory thresholds and observable expression in et al., 2009). Sensory behaviors may be reported
populations with ASD, socio-communicative differently by the same caregiver in different
impairment may offer a logical explanation contexts or over time: parents may be most reliable
(Gilbert-MacLeod et al., 2000). Gilbert-MacLeod et in reporting behaviors that are more frequent,
al. (2000) found that developmentally delayed intense or disruptive in terms of daily activities
children were less likely to engage in help-seeking (Little et al., 2011).
behavior following a painful event compared to
non-delayed children, which may impair caregivers’ Summary and suggestions for future
ability to detect a child’s pain. It would be research
beneficial for future studies to examine help- In summary, it appears as though children with
seeking behavior following painful episodes in ASD may have difficulty integrating and
children with ASD. Aberrant responses to the social interpreting sensory experiences, but it is unclear
environment, characteristic to individuals with ASD, whether children with ASD actually express and/or
may impact pain behavior and hinder caregivers’ experience pain differently than their peers. Our
pain assessment and management (Nader et al., understanding of pain expression and experience are
2004; Inglese, 2008; Craig, 2009). Fitzgibbon and related to the characteristics of the individual as
colleagues (2013) argue that in individuals with well as the assessment method. Children with ASD
ASD, sensory pain processing abnormalities may be may not provide reliable verbal or behavioral cues
associated with impaired social processing, since for their experiences of pain, and parents of children
both physical and social pain may be atypical in this with ASD may not be reliable in their proxy reports.
population, and have overlapping neural correlates. Unfortunately it can be difficult for others to
Numerous other factors may influence our meaningfully assess pain behaviors due to their own
understanding of pain in this population including beliefs/biases, the child's idiosyncratic behavior and
cognitive functioning and presence of other socio-communicative deficits, and assessment is
psychological issues which could influence pain likely affected by context. Pain assessment, which
(Coll et al., 2011). There is considerable variability is foundational to understanding pain experience
in the level of cognitive functioning of individuals and guiding treatment, remains a challenge.
with ASD and in research samples. For example, Mistaken beliefs about pain sensitivity may lead to
some research samples (e.g. Militerni et al., 2000; inadequate treatment of pain for children with ASD.
Nader et al., 2004; Tordjman et al., 2009) included Malviya et al. (2001) found that following surgery,
low-moderate functioning individuals whereas pain assessment occurred less frequently for
Bandstra et al. (2012)’s sample was in the high children with cognitive impairment, who also
functioning range. Research sample characteristics received less pain medication; while the authors
also have clear implications for the generalizability speculated on a number of reasons for these
of the findings. Unfortunately, the current state of findings including the potential of differential pain
the literature does not allow conclusions about perception, it could have also been due to

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clinicians’ beliefs of altered pain perceptions of population the particular findings are
individuals with cognitive impairment. Future generalizable.
studies should examine these issues in a sample
with ASD. It is recommended that clinicians use
Much more systematic research with multimodal measures of pain (e.g. parent proxy,
populations with ASD is needed before solid self-report, observational, physiological) when
conclusions about the interrelationships of pain conducting pain assessments with this population.
sensitivity, experience, expression, and behaviour Furthermore, it is important that caregivers be
can be made. Future research should: particularly aware of painful events in individuals
x Examine pain sensitivity using multimodal with ASD (Tordjman et al., 2009), since altered
assessment including brain imaging pain behavior/expression does not mean that there is
technologies such as functional magnetic altered pain sensitivity, experience or harm.
resonance imaging (fMRI) or transcranial Amanda K. I. Knoll
magnetic stimulation (TMS) (Fitzgibbon et al., University of Guelph, Guelph, ON, Canada
2013);
C. Meghan McMurtry, PhD, CPsych
x Directly compare children with ASD with other
Department of Psychology, University of Guelph,
populations;
Guelph, ON, Canada
x Examine the relationships among caregiver
email: cmcmurtr@[Link]
beliefs, their day-to-day experience with their
child, and pain ratings for their own child vs. Christine T. Chambers, PhD, RPsych
others with ASD; Departments of Pediatrics, Psychology, Anesthesia
x Control for cognitive impairment to understand and Psychiatry, Dalhousie University, Halifax, NS,
its contributions to the issue while also Canada
considering to which subgroups of the ASD

Endnote
1
Hyposensitivity, rather than hypersensitivity, has been emphasized in successive versions of the DSM, and
also in the Sensory Experiences Questionnaire developed by Baranek et al. (2006) which contains an item
asking about pain hyposensitivity only. The physiological mechanisms underlying pain and self-injurious
behaviors in individuals with ASD and other developmental disabilities, such as differences in the release of
endogenous opioids, are beyond the scope of this commentary (see Oswald et al., 1994, Canitano, 2006,
Symons, 2011).
References
American Psychiatric Association. Diagnostic and Baranek GT. Efficacy of sensory and motor
statistical manual of mental disorders (5th ed.). interventions for children with autism. J Autism Dev
Washington, DC: Author, 2013. Disord 2002;32:397-422. [Link]/12463517
[Link]/oclc/830807378
Baranek GT, David FJ, Poe MD, Stone WL, Watson LR.
Ashburner J, Ziviani J, Rodger S. Sensory processing Sensory Experiences Questionnaire: discriminating
and classroom emotional, behavioral, and educational sensory features in young children with autism,
outcomes in children with autism spectrum disorder. Am developmental delays, and typical development. J Child
J Occup Ther 2008;62:564-573. Psychol Psychiatry 2006;47:591-601.
[Link]/18826017 [Link]/16712636
Bandstra NF, Johnson SA, Filliter JH, Chambers CT. Ben-Sasson A, Hen L, Fluss R, Cermak SA, Engel-
Self-reported and parent-reported pain for common Yeger B, Gal E. A meta-analysis of sensory modulation
painful events in high-functioning children and symptoms in individuals with autism spectrum disorders.
adolescents with autism spectrum disorder. Clin J Pain J Autism Dev Disord 2009;39:1-11.
2012;28:715-721. [Link]/22699139 [Link]/18512135

26
Pediatric Pain Letter, August 2013, Vol. 15 No. 2 [Link]/ppl

Bottos S, Chambers CT. The epidemiology of pain in Fitzgibbon BM, Segrave RA, Fitzgerald PB, Enticott PG.
developmental disabilities. In: Symons F, Oberlander T, Can studies of pain help to bridge the gap between
editors. Pain in children and adults with developmental sensory and social impairments in autism? Front Hum
disabilities. Baltimore: Paul H. Brookes, 2006. pp. 67-87. Neurosci 2013;7:103. [Link]/23544015
[Link]/oclc/65361312
Gilbert-MacLeod CA, Craig KD, Rocha EM, Mathias
Breau LM, Burkitt C. Assessing pain in children with MD. Everyday pain responses in children with and
intellectual disabilities. Pain Res Manag 2009;14:116- without developmental delays. J Pediatr Psychol
120. [Link]/19532853 2000;25:301-308. [Link]/10880060
Breau LM, MacLaren J, McGrath PJ, Camfield CS, Güçlü B, Tanidir C, Mukaddes NM, Unal F. Tactile
Finley AG. Caregivers’ beliefs regarding pain in sensitivity of normal and autistic children. Somatosens
children with cognitive impairment: relation between Mot Res 2007;24:21-33. [Link]/17558920
pain sensation and reaction increases with severity of
impairment. Clin J Pain 2003;19:335-344. Hadjistavropoulos T, von Baeyer C, Craig KD. Pain
[Link]/14600533 assessment in persons with limited ability to
communicate. In: Turk DC, Melzack R, editors.
Breau LM, McGrath PJ, Zabalia M. Assessing pediatric Handbook of pain assessment (2nd ed.). New York:
pain and developmental disabilities. In: Symons F, Guilford Press, 2001. pp. 134-149.
Oberlander T, editors. Pain in children and adults with [Link]/oclc/45836036
developmental disabilities. Baltimore: Paul H. Brookes,
2006. pp. 149-172. [Link]/oclc/65361312 Inglese MD. Pain expression in children with autism
spectrum disorder (ASD): a foundation for instrument
Canitano R. Self injurious behavior in autism: clinical development. University of Florida, unpublished PhD
aspects and treatment with risperidone. J Neural Transm Thesis, 2008.
2006;113:425-431. [Link]/16075185
Klintwall L, Holm A, Eriksson M, Carlsson LH, Olsson
Cascio C, McGlone F, Folger S, Tannan V, Baranek G, MB, Hedvall A, et al. Sensory abnormalities in autism: a
Pelphrey KA, et al. Tactile perception in adults with brief report. Res Dev Disabil 2011;32:795-800.
autism: a multidimensional psychophysical study. J [Link]/21111574
Autism Dev Disord 2008;38:127-137.
[Link]/17415630 Little LM, Freuler AC, Houser MB, Guckian L, Carbine
K, David FJ, et al. Psychometric validation of the
Coll MP, Gregoire M, Latimer M, Eugene F, Jackson PL. Sensory Experiences Questionnaire. Am J Occup Ther
Perception of pain in others: implications for caregivers. 2011;65:207-210. [Link]/21476368
Pain Manag 2011;1:257-265.
Malviya S, Voepel-Lewis T, Tait AR, Merkel S, Lauer
Craig KD. Social modeling influences: pain in context. A, Munro H, et al. Pain management in children with
In: Sternbach RA, editor. The psychology of pain (2nd and without cognitive impairment following spine fusion
ed.). New York: Raven Press, 1986. pp. 67-95. surgery. Paediatr Anaesth 2001;11:453-458.
[Link]/oclc/299407109 [Link]/11442864
Craig KD. The social communication model of pain. McPartland JC, Reichow B, Volkmar FR. Sensitivity
Can Psychol 2009;50:22-32. and specificity of proposed DSM-5 diagnostic criteria
for autism spectrum disorder. J Am Acad Child Adolesc
Dickie VA, Baranek GT, Schultz B, Watson LR, Psychiatry 2012;51:368-383.
McComish CS. Parent reports of sensory experiences of [Link]/22449643
preschool children with and without autism: a qualitative
study. Am J Occup Ther 2009;63:172-181. Messmer RL, Nader R, Craig KD. Brief report: judging
[Link]/19432055 pain intensity in children with autism undergoing
venepuncture: the influence of facial activity. J Autism
Fanurik D, Koh JL, Schmitz ML, Harrison RD, Conrad Dev Disord 2008;38:1391-1394.
TM. Children with cognitive impairment: parent report [Link]/18161016
of pain and coping. J Dev Behav Pediatr 1999;20:228-
234. [Link]/10475597 Militerni R, Bravaccio C, Falco C, Puglisi-Allegra S,
Pascucci T, Fico C. Pain reactivity in children with
autistic disorder. J Headache Pain 2000;1:53-56.

27
Pediatric Pain Letter, August 2013, Vol. 15 No. 2 [Link]/ppl

Minshew NJ, Hobson JA. Sensory sensitivities and Talay-Ongan A, Wood K. Unusual sensory sensitivities
performance on sensory perceptual tasks in high- in autism: a possible crossroads. Int J Disab Dev
functioning individuals with autism. J Autism Dev Educ;47:201-212.
Disord 2008;38:1485-1498.
[Link]/18302014 Tomchek SD, Dunn W. Sensory processing in children
with and without autism: a comparative study using the
Nader R, Oberlander TF, Chambers CT, Craig KD. short sensory profile. Am J Occup Ther 2007;61:190-
Expression of pain in children with autism. Clin J Pain 200. [Link]/17436841
2004;20:88-97. [Link]/14770048
Tordjman S, Anderson GM, Botbol M, Brailly-Tabard S,
Oberlander TF, Symons FJ. An introduction to the Perez-Diaz F, Graignic R, et al. Pain reactivity and
problem of pain in developmental disability. In: Symons plasma beta-endorphin in children and adolescents with
F, Oberlander T, editors. Pain in children and adults with autistic disorder. PLoS One 2009;4:e5289.
developmental disabilities. Baltimore: Paul H. Brookes, [Link]/19707566
2006. pp. 1-4. [Link]/oclc/65361312
van Steensel FJ, Bögels SM, Perrin S. Anxiety disorders
Oswald DP, Ellis CR, Singh NN. Self-injury. In: Matson in children and adolescents with autistic spectrum
JL, editor. Autism in children and adults: etiology, disorders: a meta-analysis. Clin Child Fam Psychol Rev
assessment, and intervention. Pacific Grove, CA: 2011;14:302-317. [Link]/21735077
Brooks/Cole. pp. 147-164.
[Link]/oclc/27435078 Watling RL, Deitz J, White O. Comparison of Sensory
Profile scores of young children with and without autism
Symons FJ. Self-injurious behavior in spectrum disorders. Am J Occup Ther 2001;55:416-423.
neurodevelopmental disorders: relevance of nociceptive [Link]/11723986
and immune mechanisms. Neurosci Biobehav Rev
2011;35:1266-1274. [Link]/21237197

28